Sagliker Syndrome: A Case Series and Literature Review With Clinical Characteristics, Post Parathyroidectomy Biochemical Trajectories and Hungry Bone Syndrome

OBJECTIVE: To describe the clinical, biochemical, radiological, histopathological, and postoperative characteristics of patients with Sagliker syndrome. METHODS: Three patients with dialysis-dependent end-stage renal disease and Sagliker syndrome who underwent parathyroidectomy at our institution were retrospectively analysed. A literature review identified six published reports with detailed clinical, biochemical, radiological, surgical, and postoperative data suitable for comparative analysis. RESULTS: All three patients presented with progressive diffuse craniofacial skeletal remodelling characterised by maxillomandibular enlargement, facial widening, and severe secondary hyperparathyroidism. Preoperative intact parathyroid hormone concentrations ranged from 2258 to 4750 pg/mL, accompanied by marked hyperphosphatemia and elevated alkaline phosphatase levels consistent with advanced high-turnover bone disease. Sequential clinical imaging demonstrated progression from preserved facial morphology to severe craniofacial deformity following end-stage renal disease. Radiologic findings included osteitis fibrosa cystica, acro-osteolysis, and salt-and-pepper skull appearance. All patients developed severe postoperative hungry bone syndrome requiring intensive calcium and calcitriol supplementation following parathyroidectomy. Published reports demonstrated a highly concordant results with prolonged dialysis exposure, female predominance, severe biochemical hyperparathyroidism, diffuse craniofacial involvement, and frequent postoperative hypocalcemia. CONCLUSIONS: Early recognition and optimal management of CKD-MBD is pivotal in preventing severe, irreversible complications, including craniofacial deformities.

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Journal
Clinical Endocrinology
Published
2026-10-27
DOI
https://doi.org/10.1111/cen.70226
Primary Topic
Parathyroid Disorders and Treatments
Type
article
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article

Sagliker Syndrome: A Case Series and Literature Review With Clinical Characteristics, Post Parathyroidectomy Biochemical Trajectories and Hungry Bone Syndrome

Sanjay Kumar Bhadada, Rimesh Pal, Divya Dahiya, Uma Nahar Saikia et al.
1 links
Clinical Endocrinology
Parathyroid Disorders and Treatments
article

Sagliker Syndrome: A Case Series and Literature Review With Clinical Characteristics, Post Parathyroidectomy Biochemical Trajectories and Hungry Bone Syndrome

Sanjay Kumar Bhadada, Rimesh Pal, Divya Dahiya, Uma Nahar Saikia, Vinitaa Jha, Amal Shaharyar, Soham Mukherjee, Siddharth Madhad, Puneet Bhardwaj
article en

Abstract

OBJECTIVE: To describe the clinical, biochemical, radiological, histopathological, and postoperative characteristics of patients with Sagliker syndrome. METHODS: Three patients with dialysis-dependent end-stage renal disease and Sagliker syndrome who underwent parathyroidectomy at our institution were retrospectively analysed. A literature review identified six published reports with detailed clinical, biochemical, radiological, surgical, and postoperative data suitable for comparative analysis. RESULTS: All three patients presented with progressive diffuse craniofacial skeletal remodelling characterised by maxillomandibular enlargement, facial widening, and severe secondary hyperparathyroidism. Preoperative intact parathyroid hormone concentrations ranged from 2258 to 4750 pg/mL, accompanied by marked hyperphosphatemia and elevated alkaline phosphatase levels consistent with advanced high-turnover bone disease. Sequential clinical imaging demonstrated progression from preserved facial morphology to severe craniofacial deformity following end-stage renal disease. Radiologic findings included osteitis fibrosa cystica, acro-osteolysis, and salt-and-pepper skull appearance. All patients developed severe postoperative hungry bone syndrome requiring intensive calcium and calcitriol supplementation following parathyroidectomy. Published reports demonstrated a highly concordant results with prolonged dialysis exposure, female predominance, severe biochemical hyperparathyroidism, diffuse craniofacial involvement, and frequent postoperative hypocalcemia. CONCLUSIONS: Early recognition and optimal management of CKD-MBD is pivotal in preventing severe, irreversible complications, including craniofacial deformities.

Clinical Endocrinology
Post Graduate Institute of Medical Education and Research (IN)
Good health and well-being
Openalex Percentile: Top 12%
Parathyroid Disorders and Treatments
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